Ciliary kinesins beyond IFT: Cilium length, disassembly, cargo transport and signalling

Reilly, Madeline Louise; Benmerah, Alexandre

doi:10.1111/boc.201800074

Cited by 29 publications

(25 citation statements)

References 138 publications

(179 reference statements)

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“…Here, we report on a new role of kinesin KIF14 in primary cilium formation. Kinesins are molecular motors capable of binding microtubules through their motor domain, and they are involved in various aspects of intracellular transport, including ciliogenesis (Hirokawa et al, 2009;Reilly and Benmerah, 2019;Silverman and Leroux, 2009). They may serve different functions in primary cilia.…”

Section: Introductionmentioning

confidence: 99%

KIF14 controls ciliogenesis via regulation of Aurora A and is important for Hedgehog signaling

Pejšková

Reilly

Binó

et al. 2020

Journal of Cell Biology

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Primary cilia play critical roles in development and disease. Their assembly and disassembly are tightly coupled to cell cycle progression. Here, we present data identifying KIF14 as a regulator of cilia formation and Hedgehog (HH) signaling. We show that RNAi depletion of KIF14 specifically leads to defects in ciliogenesis and basal body (BB) biogenesis, as its absence hampers the efficiency of primary cilium formation and the dynamics of primary cilium elongation, and disrupts the localization of the distal appendage proteins SCLT1 and FBF1 and components of the IFT-B complex. We identify deregulated Aurora A activity as a mechanism contributing to the primary cilium and BB formation defects seen after KIF14 depletion. In addition, we show that primary cilia in KIF14-depleted cells are defective in response to HH pathway activation, independently of the effects of Aurora A. In sum, our data point to KIF14 as a critical node connecting cell cycle machinery, effective ciliogenesis, and HH signaling.

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Section: Introductionmentioning

confidence: 99%

KIF14 controls ciliogenesis via regulation of Aurora A and is important for Hedgehog signaling

Pejšková

Reilly

Binó

et al. 2020

Journal of Cell Biology

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“…Cilium disassembly occurs in a biphasic manner, with the first major wave during G1 shortly after mitogen stimulation and the second wave prior to mitosis [67]. Dissection of cilium disassembly in different cell types and organisms together described the physical events that underlie primary cilium disassembly, which include resorption of the ciliary axoneme to the cell body, progressive shortening of the cilium and excision of all or parts of the cilium [49,61,[67][68][69][70][71]. A recent paper by Mirvis et al 2019 used live imaging to visualize different steps of cilium disassembly in kidney epithelial cells, which uncovered heterogeneity in its dynamics and mechanisms [72].…”

Section: Primary Ciliummentioning

confidence: 99%

A Proximity Mapping Journey into the Biology of the Mammalian Centrosome/Cilium Complex

Arslanhan

Gulensoy

Fırat-Karalar

2020

Cells

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The mammalian centrosome/cilium complex is composed of the centrosome, the primary cilium and the centriolar satellites, which together regulate cell polarity, signaling, proliferation and motility in cells and thereby development and homeostasis in organisms. Accordingly, deregulation of its structure and functions is implicated in various human diseases including cancer, developmental disorders and neurodegenerative diseases. To better understand these disease connections, the molecular underpinnings of the assembly, maintenance and dynamic adaptations of the centrosome/cilium complex need to be uncovered with exquisite detail. Application of proximity-based labeling methods to the centrosome/cilium complex generated spatial and temporal interaction maps for its components and provided key insights into these questions. In this review, we first describe the structure and cell cycle-linked regulation of the centrosome/cilium complex. Next, we explain the inherent biochemical and temporal limitations in probing the structure and function of the centrosome/cilium complex and describe how proximity-based labeling approaches have addressed them. Finally, we explore current insights into the knowledge we gained from the proximity mapping studies as it pertains to centrosome and cilium biogenesis and systematic characterization of the centrosome, cilium and centriolar satellite interactomes.

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“…KIF7 is a ciliary kinesin, which plays a crucial and conserved role in the organisation of a SHH signalling platform at the distal end of PC [Bangs and Anderson, ; Reilly and Benmerah, ]. In both humans and mice, KIF7 depletion leads to overactivation of the SHH pathway associated to abnormal GLI3 processing [Liem et al., ; Putoux et al., ].…”

Section: Ciliopathy‐associated Cerebral Anomaliesmentioning

confidence: 99%

Cilia in hereditary cerebral anomalies

Thomas

Boutaud

Reilly

et al. 2019

Biology of the Cell

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Ciliopathies are complex genetic multi-system disorders causally related to abnormal assembly or function of motile or non-motile cilia. While most human cells possess a non-motile sensory/primary cilium (PC) during development and/or in adult tissues, motile cilia are restricted to specialised cells. As a result, PC-associated ciliopathies are characterised by high phenotypic variability with extensive clinical and genetic overlaps. In the present review, we have focused on cerebral developmental anomalies, which are commonly found in PC-associated ciliopathies and which have mostly been linked to Hedgehog signalling defects. In addition, we have reviewed emerging evidence that PC dysfunctions could be directly or indirectly involved in the mechanisms underlying malformations of cerebral cortical development including primary microcephaly.

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Ciliary kinesins beyond IFT: Cilium length, disassembly, cargo transport and signalling

Cited by 29 publications

References 138 publications

KIF14 controls ciliogenesis via regulation of Aurora A and is important for Hedgehog signaling

KIF14 controls ciliogenesis via regulation of Aurora A and is important for Hedgehog signaling

A Proximity Mapping Journey into the Biology of the Mammalian Centrosome/Cilium Complex

Cilia in hereditary cerebral anomalies

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